Spinal epidural abscess is an uncommon disease that accounts for 0.2–1.2 cases per 10,000 hospital admissions, with a relatively high rate of associated morbidity and mortality [2, 4, 5]. Most patients with spinal epidural abscess have systemic predisposing conditions, such as diabetes mellitus, renal disorder, human immunodeficiency virus infection, malignancy, morbid obesity, long-term corticosteroid use, alcoholism, or a distant site of infection [2, 6]. Some local conditions are also known as predisposing factors, including spine trauma, spinal surgery, and extrathecal injection or catheter placement into the vertebral canal [2, 6]. In terms of neurologic symptoms and its progression, spinal epidural abscess is divided into four stages: stage 1, back pain, fever, and local tenderness at the level of the affected spine; stage 2, signs of spinal irritation and neck stiffness; stage 3, motor weakness, sensory deficit, and bladder and bowel dysfunction; and stage 4, complete paralysis [7].
Because epidural infection can injure the spinal cord either directly by mechanical compression or indirectly as a result of vascular occlusion caused by septic thrombophlebitis, the associated neurologic dysfunction and mortality are high [6]. Especially in severe cases with neurologic impairment, such as stage 3 or 4 disease, decompressive laminectomy and debridement of infected tissues should be performed immediately [2, 4–6]. Although laminectomy is recommend for the severe case with neurologic impairment, it is impractical to perform decompressive laminectomy along with all spinal epidural abscess because of its invasiveness. Therefore, less extensive surgery might be considered for stage 1 or 2 patients with neurologically mild symptoms [6]. In this case, intestinal communication to spine was apparent and neurologic symptom was relatively mild and slowly progressive. Considering the invasiveness of laminectomy, sufficient drainage of abscess with defunctioning ileostomy and antibiotic therapy might be preferred for this case.
Patients with unexplained persistent or recurrent epidural infection should be carefully inspected for rare sources of infection. In the case of thoracolumbar abscesses, an intestinal–spinal fistula may be causative [6].
Inflammatory bowel disease, especially Crohn’s disease, is complicated by perforation, abscess, and fistula in approximately 30–50 % of cases [8, 9]. Fistula associated with Crohn’s disease sometimes develops between the intestine and other organs [10–12]. In some cases, a fistula from the intestine extends to the epidural space to form a spinal epidural abscess [13–17].
In ulcerative colitis, fistula formation is relatively rare. However, 5–10 % of cases after IPAA have been reported to develop pouch-related fistula [1, 3, 18]. Only one case of ulcerative colitis with IPAA-associated spinal epidural abscess has been previously reported. In this patient reported by Brown et al. [14], although spinal epidural abscess was precisely diagnosed and immediate laminectomy was performed, the infectious source was not detected. Only 3 years later was a fistula extending from the top of the ileal pouch to the presacral space detected. The patient underwent a combined abdominal/perineal pouch excision, and reconstruction of a new pouch with hand-sewn ileal pouch anal anastomosis and construction of a diverting ileostomy.
In the present case, we precisely diagnosed a spinal epidural abscess resulting from an ileal pouch spinal fistula, and assessed the neurologic impairment as stage 2 disease. We performed diverting ileostomy, with thorough drainage and debridement of the abscess. At the initial operation, we mainly intended to eradicate the epidural abscess by fecal diversion. At the second operation, although a presacral residual abscess had been observed, the spinal epidural abscess was cured completely. In line with a previous report that ileal pouch-related pelvic sepsis might be refractory to various treatments [19], we also experienced recurrence of the pelvic abscess. Although reoperation for drainage was required, the patient was finally able to achieve stoma closure.
To the best of our knowledge, this is the first case in the English literature of spinal epidural abscess attributable to an enteroepidural fistula arising from the ileal pouch while retaining intestinal continuity. Although pouch-related spinal epidural abscess is an extremely rare complication of ulcerative colitis after IPAA, one should include this condition in the differential diagnosis when encountering patients with systemic inflammation and neurologic impairment.
